HERLYN-WERNER WÜNDERLICH OHVIRA

SYNDROME (OBSTRUCTED HEMI-VAGINA AND

IPSILATERAL RENAL ANOMALY), DIFFERENT

FORMS OF CLINICAL PRESENTATION AND HOW TO

INTEGRATE 2D AND 3D ULTRASOUND FOR PROPER

MANAGEMENT.

CASE SERIES

SÍNDROME DE HERLYN-WERNER WÜNDERLICH OHVIRA

(HEMIVAGINA OBSTRUIDA Y ANOMALÍA RENAL

IPSILATERAL), DIFERENTES FORMAS DE PRESENTACIÓN

CLÍNICA Y CÓMO INTEGRAR EL ULTRASONIDO 2D Y 3D

PARA UN MANEJO ADECUADO.

SERIES DE CASOS

Juan Carlos Quintero

Universitary Hospital del Valle Evaristo García

John Jairo Rojas

Libre University of Cali

Natalia Quintero

Sntiago de Cali University

pág. 3727

DOI: https://doi.org/10.37811/cl_rcm.v8i3.11586

Herlyn-Werner Wünderlich Ohvira Syndrome (Obstructed Hemi-Vagina

And Ipsilateral Renal Anomaly), Different Forms of Clinical Presentation

and How to Integrate 2d and 3d Ultrasound for Proper Management.

Case Series

Juan Carlos Quintero

juancarlosquintero63@gmail.com

https://orcid.org/0009-0008-9732-1367

Clínica Imbanaco

Quirón Salud Group, Valle University

Universitary Hospital del Valle Evaristo García

John Jairo Rojas

jrojaspedraza@gmail.com

https://orcid.org/0009-0006-4645-0770

Farallones de Cali Clinic

Libre University of Cali

Natalia Quintero

nqs90@hotmail.com

https://orcid.org/0009-0007-6214-5976

Clínica imbanaco grupo Quirón salud

Sntiago de Cali University

ABSTRACT

Introduction: Obstructed hemivagina and ipsilateral renal anomaly (OHVIRA) syndrome, is a rare

Mullerian duct anomaly with uterus didelphys, unilateral obstructed hemivagina, and ipsilateral renal

agenesis. In most cases, Patients who rarely present this structural anomaly, start to experience

sympotms such as chronic pelvic pain and mass sensation that usually swap that star wirh menarche

and variate with the menstrual cycle. Objective: this is an original article which aims to describe the

wide range of symptoms presented by patients with (OHVIRA) syndrome, how their quality of life is

affected due to underdiagnosis, and how 2D and 3D ultrasound is an important tool for the diagnosis of

an adequate surgical technique. Matherials and methods: narrative describes the case of 2 adolescent

patients who were incidentally diagnosed with the syndrome in the city of Cali Colombia at the

imbanaco Clinic. information was obtained from their respective clinical histories, and ultrasound

images and the respective surgical techniques used for the management of the symptoms were also

extracted. the informed consent of both patients was obtained. Results: The diagnosis is usually late or

incidental because the clinical presentation only becomes evident once menarche occurs. As the clinical

and structural presentation of the pathology comprises a wide spectrum, therefore the diagnosis today

is made by means of 2D and 3D ultrasonography, diagnostic imaging such as computed axial

tomography and magnetic resonance imaging. the management will depend on the structural anomaly

and the intensity of the symptoms. Today surgical method are usually the treatment of choice.

Conclusion: OHVIRA syndrome is a rare congenital anomaly that represents a diagnostic challenge

because of the regular menstruation and nonspecific clinical presentation . Health care providers should

be aware of the different extending symptoms in order to employ different diagnose images such as 2D

and 3D ultrasonography operated by highly trained staff (sensibility >90 % y specificity 100% to

facilitate early and accurate management and improve patients quality life.

Keywords : uterine didelphys, solitary kidney, ultrasonography, hematocolpos, quality of life

Autor principal

Correspondencia: juancarlosquintero63@gmail.com

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Síndrome de Herlyn-Werner Wünderlich Ohvira (Hemivagina Obstruida y

Anomalía Renal Ipsilateral), Diferentes Formas de Presentación Clínica y

Cómo Integrar el Ultrasonido 2d y 3d para un Manejo Adecuado.

Series de Casos

RESUMEN

Introducción: El síndrome de hemivagina obstruida y anomalía renal ipsilateral (OHVIRA), es una rara

anomalía del conducto de Müller con útero didelfo, hemivagina obstruida unilateral y agenesia renal

ipsilateral. En la mayoría de los casos, las pacientes que rara vez presentan esta anomalía estructural,

comienzan a experimentar síntomas como dolor pélvico crónico y sensación de masa que suele

intercambiar esa estrella con la menarquia y variar con el ciclo menstrual. Objetivo: este es un artículo

original que tiene como objetivo describir la amplia gama de síntomas que presentan los pacientes con

síndrome (OHVIRA), cómo su calidad de vida se ve afectada por el infradiagnóstico y cómo la ecografía

2D y 3D es una herramienta importante para el diagnóstico del síndrome. una técnica quirúrgica

adecuada. Materiales y métodos: se describe narrativamente el caso de 2 pacientes adolescentes que

fueron diagnosticados incidentalmente con el síndrome en la ciudad de Cali Colombia en la Clínica

Imbanaco. Se obtuvo información de sus respectivas historias clínicas, así como también se extrajeron

imágenes ecográficas y las respectivas técnicas quirúrgicas utilizadas para el manejo de los síntomas.

Se obtuvo el consentimiento informado de ambos pacientes. Resultados: El diagnóstico suele ser tardío

o incidental porque la presentación clínica sólo se hace evidente una vez que ocurre la menarquia. Como

la presentación clínica y estructural de la patología comprende un amplio espectro, hoy en día el

diagnóstico se realiza mediante ecografía 2D y 3D, diagnóstico por imágenes como la tomografía axial

computarizada y la resonancia magnética. el manejo dependerá de la anomalía estructural y de la

intensidad de los síntomas. Hoy en día el método quirúrgico suele ser el tratamiento de elección.

Conclusión: El síndrome OHVIRA es una anomalía congénita poco común que representa un desafío

diagnóstico debido a la regularidad de la menstruación y la presentación clínica inespecífica. Los

proveedores de atención médica deben ser conscientes de los diferentes síntomas que se extienden para

emplear diferentes imágenes de diagnóstico, como la ecografía 2D y 3D operada por personal altamente

capacitado (sensibilidad >90 % y especificidad 100 % para facilitar un manejo temprano y preciso y

mejorar la calidad de vida de los pacientes.

Palabras clave: didelfo uterino, riñón único, ecografía, hematocolpos, calidad de vida

Artículo recibido 20 abril 2024

Aceptado para publicación: 25 mayo 2024

pág. 3729

INTRODUCTION

HERLYN-WERNER-WÜNDERLICH (OHVIRA) syndrome is a congenital urogenital malformation

characterized by the presence of an obstructed hemivagina, a didelphic uterus and ipsilateral renal

agenesis. This syndrome is a rare congenital malformation caused by abnormal development of the

Müllerian and Wolffian ducts during embryonic development of the female reproductive system, is very

rare, with an undetermined prevalence due to clinical underdiagnosis in many cases; representing less

than 10% of all Müllerian malformations; the exact rate is unknown but is thought to range from 1/2000

to 1/28000 women.

Although its pathophysiology is not very clear, it is believed that this type of Müllerian malformation

may be the final product of a deficit in the fusion, canalization and reabsorption of the septum of the

Müllerian (paramesonephric) and Wolf (mesonephric) ducts. In most cases (30-50%) there are usually

concomitant genitourinary anomalies, due to the joint embryonic development of both systems during

the early stages of human embryogenesis, which leads to the coexistence of genital and urological

malformations.

2,3

The spectrum of symptoms is usually very broad and will depend on the % of vaginal obstruction,

however, in most cases patients experience dysmenorrhea and progressive pelvic pain which are usually

the cardinal symptoms. Diagnostic delays are frequent, and are mainly due to the presence of patients

with regular menstrual cycles.

4,5,6

The European Society of Human Reproduction and Embryology in conjunction with the European

Society of Gynecological Endoscopy has generated a classification that describes the type of uterine,

cervical and vaginal abnormality in order to offer the most appropriate management for each patient.

1,4,7

(Table 1).

pág. 3730

Table 1

Grimbizis, G.F., Gordts, S., Di Spiezio Sardo, A., Brucker, S.Y., De Angelis, C., Gergolet, M., Li, T.C., Tanos, V., Brölmann,

H., Gianaroli, L., & Campo, R. (2013). The ESHRE/ESGE consensus on the classification of female genital tract congenital

anomalies. Human Reproduction (Oxford, England), 28, 2032 - 2044.

This article describes 2 cases presented in the city of Cali, Colombia in patients of different age groups,

with a wide spectrum of symptoms which were diagnosed through the use of gynecologic

ultrasonography operated by highly skilled specialists who contributed to a timely diagnosis. The

patients are currently undergoing various medical and surgical treatments with a positive prognosis.

8,9

Narrative

Case 1

The first case corresponds to a 20-year-old female university student, previously diagnosed with

congenital renal agenesis since birth in continuous management with nephrology, pregnant 0 para 0,

who first consulted the gynecology service at 17 years of age, referred from the emergency department

for presenting a clinical picture of approximately 3 months of evolution, consisting of progressive pelvic

pain of uncertain etiology associated with dysmenorrhea which did not yield to the use of

pág. 3731

antispasmodics or analgesics, the patient referred regular menstrual cycles. Therefore, with the

suspicion of a possible appendicitis, which was ruled out, it was decided to refer the patient to the

gynecology and obstetrics service, where a transvaginal gynecological ultrasound was performed,

which reported a complex cyst adjacent to the cervix and it was suggested to complement it with

computed axial tomography or abdomino-pelvic magnetic resonance imaging.

The patient was referred to a specialist in gynecology and obstetrics where the attending gynecologist

decided to take the patient to an exploratory laparoscopy with hysteroscopy, where resection of the right

hemiuterus and right salpingectomy were performed. (image 1) the operative findings report a vagina

without lesions, a single cervix is observed, a 5-6 cm mass is palpated which ablates the posterior fundus

lateral to the right cervix (image 2), the only visible ostium is observed in the left cavity, the left

hemiuterus looks normal, the right hemiuterus has an elongated shape with hematometra extending

from the endometrial cavity to the right endocervix, the fallopian tubes are normal. Normal ovaries.

Normal tubes. Resection of the right hemiuterus and salpingectomy (image 3), biopsy of uterine tissue

without histological abnormalities with diagnosis of right hemiuterus, proliferative endometrium,

myometrium without particularities and uterine tuba with foci of old hemorrhage.

Image 1 Speculoscopy showing on the right side of the patient the transverse vaginal septum without

cervical os and on the right side the cervix of the left hemisphere.

pág. 3732

Image 2: upper laparoscopic view showing a kind of mass attached to the uterus on the right side of the

patient, which is recognized as the right hemi uterus with collection (hematocolpos).

Image 3: Resection of right hemiuterus and right salpingectomy by laparoscopy and hysteroscopy.

After surgery, the specialist sent a pelvic MRI that reported failure in the fusion of the mullerian ducts,

with a didelphic uterus with obstruction of the cervical orifice on the right side and a hematic collection

in its interior (image 4).

pág. 3733

Image 4

Animated illustration showing the surgery performed (exploratory laparoscopy with hysteroscopy,

where resection of the right hemiuterus is performed, right salpingectomy) source of elaboration

Gabriela Quintero Rodríguez, illustrator.

Therefore, he considers that the most appropriate thing to do is to refer the patient to a highly qualified

specialist in ultrasound diagnosis, where a 2D transvaginal ultrasound is performed, which reports a

normal echogenicity left hemi uterus, measuring 70x32x41 mm with volume 49cc, endometrial echo of

7.9mm menstruating. In the right hemivagina an oval image measuring 49 x 32 mm with ground glass

content corresponding to hemicervix with hematocolpos is seen (Image 5). A 2D and 3D reconstruction

of the initial ultrasound image is performed (Image 5 and 6). There is marked sensitivity expressed by

the patient during the exploration. It is important to emphasize that at this time the patient had already

been admitted for surgery and therefore only a uterine cavity with adnexal vaginal mass is observed,

thus confirming ultrasonographically the previous diagnosis of OHVIRA U3(b) C2 V2 syndrome

(Image 7).

pág. 3734

Image 5: transvaginal pelvic ultrasound showing on the right side of the image a collection of dense

echo content and on the left side a hemi cervix without alterations.

Image 6: three-dimensional reconstruction of the previous image showing a collection on the right side

and an unaltered hemi cervix on the left side.

pág. 3735

Image 7: three-dimensional reconstruction of the residual left hemiuterus with normal appearance

(image taken after surgery).

To date, the patient reports that after the first surgery she felt improvement of the mentioned

symptomatology, however, approximately two months ago she has presented the same clinical picture

again, reason for which she is waiting for a trachelectomy by laparoscopy in order to definitively

improve her quality of life and the possibility of possible complications.

Case 2

This is a 12-year-old female patient with no personal or family pathological history who consults the

emergency department of a hospital in the city, with no personal or family pathological history, who

consults for presenting a clinical picture of 3 days of evolution in the lumbar portion with irradiation to

the right inguinal area, which did not subside with oral analgesics. In the hospital a diagnosis of renal

lithiasis vs. appendicitis was made. A renal and urinary tract ultrasound was performed, reporting a left

kidney not visualized in the left lumbar fossa or the rest of the abdomen, uterus of usual ultrastructure,

with a mass in the projection of the left ovary suggestive of teratoma and left renal agenesis with right

kidney of compensatory aspect, suggesting a CT scan to complement the diagnosis and correlate with

clinical symptoms. CA 125, CA 19-9, carcinoembryonic antigen, alpha fetoprotein and qualitative beta

subunit chorionic gonadotropin were negative.

pág. 3736

It was decided to perform a contrasted abdominal tomography which reported left renal agenesis with

compensatory renal hypertrophy, didelphic uterus without the possibility of defining a septum with

associated vaginal collection, apparently left hemivagina. The previous findings are suggestive of

OHVIRA syndrome, and a contrasted pelvic MRI and evaluation by gynecology are suggested. The

contrasted pelvic MRI reports didelphic uterus with duplication of cervix, duplication of vaginal cavities

with distension at the expense of liquid content that could indicate a hematic nature (hematocolpos)

without ruling out a secretory (infectious) nature. Multifollicular ovaries, scarce free fluid.

The patient began to refer to her mother the presence of a yellow, fetid vaginal discharge associated

with severe pelvic pain which did not yield to the use of painkillers so she consulted the emergency

department, where a vaginal smear was performed which reported positive fungal +++ pH 6, negative

Gardenella and negative vaginal culture; She is medicated with oral metronidazole for 7 days and is

referred to consider surgical management for vaginal and uterine reconstruction at level III in order to

reduce symptoms and improve the patient's quality of life.

In consultation with a specialist, surgical intervention is suggested and transvaginal ultrasound is

requested by a highly qualified specialist in ultrasonographic diagnosis, which reports: OHVIRA

syndrome, didelphic uterus with classification U3 (b) C2, V3 (image 8 and 11), hematocolpos in left

hemivagina, vaginal septum, ipsilateral renal agenesis, a 3D virtual reconstruction is generated (image

9 and 10) and an imaging report is sent to the surgical specialist in order to guide the most opportune

decision making regarding the various possibilities of medical surgical management indicated for the

pathological characteristics of the patient..

pág. 3737

Image 8: transabdominal ultrasound in sagittal view showing left hemiuterus with collection at the level

of the left vaginal canal.

Image 9

Abdominal ultrasound with three-dimensional reconstruction where the two uterine bodies can be

differentiated bi-corporeal didelphic uterus with the presence of distal vaginal hematocolpos

corresponding to left hemiuterus.

pág. 3738

Image 10: three-dimensional reconstruction via the abdominal route showing two uterine bodies with

bilateral endometrial stimulation.

Image 11: illustration showing hematocolpos in left hemivagina, vaginal septum, ipsilateral renal

agenesis. source Gabriela Quintero Rodríguez. illustrator.

After the 2D and 3D ultrasound examination the patient is interviewed by the surgeon who after seeing

the images decides that the patient is suitable to undergo hysteroscopy in order to drain the

hematometrium, dissect the uterine septum and significantly improve her quality of life to avoid

possible complications.

pág. 3739

MATERIALS AND METHODS

A descriptive case report type study was performed, without intervention on the analysis of the two

clinical histories compatible with HERLYN-WERNER-WÜNDERLICH syndrome (OHVIRA).

The study was carried out in the city of Cali, Colombia, at the Imbanaco clinic. The respective

information was extracted from the clinical histories of the patients, highlighting the appearance of the

diversity of clinical symptoms, differential diagnoses, diagnostic methods and some treatment

alternatives. Emphasis is placed on the challenge posed by the possibility of a timely diagnosis for

health personnel. It should be noted that this case report has the approval of the ethics committee of the

Imbanaco clinic in Cali, we consider it of utmost importance to emphasize that the report in question

was made under all standards of good clinical practice and following the parameters of the Helsinki

declaration of the World Medical Association, also with the respective consent and informed assent for

the disclosure of the clinical outcome, imaging and individualized treatment of each patient.

DISCUSSION

Müllerian malformations characterized by the association of vaginal obstruction with ipsilateral renal

anomalies were first reported in the literature in 1922 by Herlyn-Werner and later in 1950 by

Wunderlich.8,10

The cases described in this report had in common the presence of a didelphic uterus, a hemivagina with

partial or total obstruction and ipsilateral renal agenesis. This characteristic diagnostic triad is now

recognized as the OHVIRA syndrome (Obstructed Hemi-vagina and Ipsilateral Renal Anomaly).

The exact incidence of this syndrome is unknown. Some reports suggest that it ranges from 1/2000 to

1/28000 women. With a prevalence of Müllerian anomalies of 7% of women of reproductive age

worldwide.1

Approximately 40% of these are associated with renal alterations. Of patients with uterine duplication,

6% have an obstructed hemivagina. Renal agenesis occurs in 63 to 81% of cases of uterine duplication,

and in 92 to 100% of cases of obstructed hemivagina.1,7,11

The exact pathophysiologic mechanisms involved in this type of Müllerian anomalies are currently

unknown. The theory of a multifactorial etiology has been proposed, including genetic and epigenetic

factors. The embryological alteration of the mesonephric or unilateral Wolffian ducts gives rise to renal

pág. 3740

agenesis. This alteration in the mesonephric duct lateralizes the ipsilateral paramesonephric Wolffian

duct, resulting in a didelphic uterus and an obstructed hemivagina.12

The diagnosis is usually made after menarche, and although its clinical spectrum is usually broad

depending on the structural anomaly and the percentage of obstruction, its classic form usually appears

as a picture of chronic pelvic pain associated or not with progressive dysmenorrhea and in some cases

with the presence of a sensation of a vaginal mass or tumor. Late diagnoses are frequent and are due to

multiple causes such as: absence of symptoms prior to menarche, progressive onset of symptoms after

the menstrual cycle, however the presence of regular menstrual cycles often distract the health

professional.

A prenatal or neonatal diagnosis can be made by exhaustive evaluation of the Müllerian structures in

fetuses or neonates diagnosed with renal anatomical alterations 13.

Diagnostic confirmation, as mentioned above, is made by means of imaging techniques both by 3D

ultrasound with image reconstruction and by MRI, the accuracy of the latter being close to 100%.8,9,12

Early diagnosis of this syndrome allows the possibility of offering the patient appropriate management

to improve her quality of life, reducing the presence of chronic pelvic pain, decreasing the appearance

of complications such as pelvic inflammatory disease, endometriosis and infertility.14

For diagnosis, 2D and 3D ultrasound and Magnetic Resonance Imaging (MRI) are useful. To obtain

images of the soft tissue anatomy and identify congenital anomalies 15. Once the diagnosis is suspected,

it is indicated to perform a tranvaginal ultrasound operated by an expert where the visualization of the

hematocolpos (if present) and the uterine malformation is allowed, 3D reconstruction ultrasound is

usually of great help for surgical planning and its operative pathway. Magnetic Resonance Imaging is

also usually of great help with a sensitivity of 100%. 8,9

It is of substantial importance to perform an exhaustive screening for Müllerian malformations in all

patients diagnosed with renal anomalies in order to generate a timely referral to the gynecology service

in order to generate a diagnosis and be able to perform an adequate management in order to improve

the quality of life of the patients.

pág. 3741

CONCLUSIONS

HERLYN-WERNER-WÜNDERLICH syndrome (OHVIRA) is a complex urogenital müllerian

anomaly, which is infrequently seen in clinical practice. Its wide variability of clinical presentation and

the lack of knowledge of it lead to a late diagnosis which ultimately increases the possibility of

presenting complications that compromise the quality of life of affected patients; altering their quality

of life and compromising their sexual and reproductive future; it is of great importance that health

professionals, from the first contact with the patient, consider the possibility of the existence of this

pathology in order to generate an early clinical diagnosis and to be able to offer the possibility of

establishing an adequate therapy; thus reducing the comorbidity related to this syndrome.

Confidentiality of data

The authors declare that they have followed with protocols of their work center on the publication of

patient data and images.

Right to privacy and informed consent

The authors declare that no patient data appear in this article. Informed consent signed by the patient is

available.

Conflicts of interest

The authors declare that they have no conflicts of interest.

Funding

The authors declare that they have not received any funding.

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